L E T T E R T O T H E E D I T O R S
High-dose praziquantel therapy for cerebral sparganosis
Roman R. Gonzenbach• Yoon Kong•Bernhard Beck•Alfred Buck•Michael Weller•
Alexander Semmler
Received: 22 January 2013 / Revised: 18 March 2013 / Accepted: 20 March 2013 / Published online: 2 April 2013 Ó Springer-Verlag Berlin Heidelberg 2013
Dear Sirs,
A 39-year-old Bangladeshi man was admitted to our hos-pital with a first generalized epileptic seizure. Brain MRI showed multiple ring-enhancing lesions with perifocal edema (Fig. 1a and g, February 2008). CSF analysis showed 8 cells/ll and normal protein, glucose and lactate levels. Serum and CSF serological tests were negative for a broad range of parasites, fungi and bacteria.
The patient was suspected to have neurocysticercosis and was treated with albendazole (400 mg bi-daily for 30 days). However, follow up MRI showed new, adjacent lesions with ring- and tunnel-shaped enhancement; and punctate calcifications in the left parieto-occipital area in the CT scan (Fig.1b, h and m, December 2009), suggesting that the treatment had been ineffective. We therefore per-formed a brain biopsy (with prior MRI for the localization of the sparganum, see Fig.1c and i, February 2010), which revealed a degenerated cyst containing membrane-shaped structures with multiple foci of calcification. PCR was
positive for Spirometra erinaceieuropaei, leading to the diagnosis of cerebral sparganosis.
Surgical removal of the parasite is the treatment of choice as standard anti-helmintic therapy is considered ineffective in sparganosis [1–3]. The repeated MRI indi-cated that the lesion had been wandering again (Fig.1d and j). An ethylcholine PET showed a defined region with increased tracer uptake, most likely indicating the current location of the sparganum (Fig.1n). However, the patient refused the recommended stereotactic surgery. We there-fore treated the patient with a high-dose regimen of pra-ziquantel (3 9 25 mg/kg body weight daily) for 7 days combined with cimetidine (3 9 400 mg daily) and a high carbohydrate diet to increase plasma levels of praziquantel [4]. The treatment was well-tolerated.
Follow-up MRIs 3 and 11 months later showed drastic improvement and the ethylcholine PET had normalized (Fig.1e, f, k, l, o and p). Anti-sparganum antibody levels were determined from preserved CSF and serum samples by a specific ELISA [5]. Before therapy (Fig.1, December 2009), anti-sparganum antibody levels were elevated in the serum (0.29) and in the CSF (0.55 normal value in serum and CSF \ 0.22). After therapy (Fig. 1, June 2011), anti-body levels had dropped to 0.09 in serum (a lumbar puncture was not repeated after therapy). Seizures dis-continued and anticonvulsive treatment was slowly tapered.
Sparganosis is a rare parasitic disease caused by infes-tation with the larval cestode of Spirometra spp. Most cases have been reported in Southeast and Eastern Asia. Humans are infected by drinking unfiltered, contaminated water or through consumption of raw snakes and frogs. When the parasite invades the central nervous system, epileptic sei-zures and headache are common symptoms [2]. Charac-teristic MRI findings of cerebral sparganosis include a R. R. Gonzenbach (&) M. Weller A. Semmler
Department of Neurology, University Hospital Zurich, Frauenklinikstrasse 26, 8091 Zurich, Switzerland e-mail: roman.gonzenbach@usz.ch
Y. Kong
Department of Molecular Parasitology, School of Medicine, Sungkyunkwan University, Seoul, Korea
B. Beck
Department of Infectiology, University Hospital, Zurich, Zu¨rich, Switzerland
A. Buck
Department of Radiology, University Hospital, Zurich, Zu¨rich, Switzerland
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J Neurol (2013) 260:1423–1425 DOI 10.1007/s00415-013-6901-7
‘tunnel sign’, and ‘wandering lesions’ on successive ima-ges representing the track of motion of the parasite [6,7]. The treatment of choice for cerebral sparganosis is surgical removal because treatment with anti-helminthics, including praziquantel, has been described as not effective [1–3]. In our patient the clinical improvement, and the normalization of brain imaging and of anti-sparganum antibody levels suggest that the parasite was eradicated by high-dose praziquantel therapy. Although the close tem-poral correlation of therapy and presumed death of the parasite suggests a causal relationship we cannot rule out the possibility that this was, in fact, due to a coinciding, natural death of the parasite and not due to the chemo-therapeutics. There is no information regarding the ces-tode’s life expectancy in human tissue in the literature.
Cerebral sparganosis should be suspected in patients from endemic areas with indications of cerebral parasitic infection and ‘‘wandering lesions’’ in the MRI. High-dose praziquantel treatment may be considered in inoperable cases of cerebral sparganosis. Ethylcholine PET can be
used preoperatively for the precise localization of the sparganum. This should be particularly valuable if the precise location is not known due to the often multifocal lesions in MRI.
Conflicts of interest On behalf of all authors, the corresponding author declares no conflict of interest.
References
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brain MRIs in chronological order. The upper row shows contrast-enhanced horizontal T1-weighted sections (a–f). The middle row shows the corresponding T2 sections (g–l). Characteristic punctate
calcifications in the CT in the affected region are shown in (m). Repeated ethylcholine PET scans before and 3 and 11 months after praziquantel therapy are shown in (n–p). The relative amount of tracer uptake is color-coded with arbitrary units
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